CervicalOsteophytosis Dysphagia: A As A Rare Cause Report [PDF]

cervical osteophytes. Aspiration is common in. Bekii: Vertebrngeiiic Dysplrngin individuals with dysphagia who have cerv

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Tiirkish

Neiirosiirgery

11: 117 -

Beieli: Vertebrnge11ic Dysphngin

120, 2001

Cervical Osteophytosis As A Rare Cause of Dysphagia: A Cas e Report Nadir Bir Disfaji

Nedeni Olan Servikal Olgu Sunumu

Osteofitozis:

DENIz BELEN, LEVENT GÜRSES

Social Security Hospital, Department of Neurosurgery,

Received: 4.11.2000

Ankara

Accepted: 10.1.2001

Abstract: Cervical osteophytes are frequently found in geriatric patients. Although it is uncommon to observe d ysphagia induced by these osteophytes, this is an important and treatable condition. The diagnosis may be established using plain radiographs of the spine, esophagography, computed tomography, or magnetic resonance imaging. We deseribe the case of a patient with severe dysphagia who had a large anterior cervical osteophyte. The differential diagnosis, treatment options, and pathophysiology of the process are discussed.

Özet: Servikal osteofitler ileri yasta sikça karsilasilan bir durumdur. Servikal osteofit sonucu ortaya çikan disfaji oldukça nadir olmasina ragmen tedavi edilebilir bir durumdur. Tani; omurga kolonunun düz grafisi, özefagogram, BT veya MRG ile konulabilir. Bu yazida servikal osteofite bagli siddetli disfajisi olan bir hasta sunularak ayiriCl tani, tedavi yöntemleri ve patofizyoloji tartisildi.

Key words: Diffuse idiopathic skeletal hyperostosis (DISH), dysphagia, Forestier's disease, spinal osteophytosis

Anahtar kelimeler: Diffüz idiopatik iskelet hiperostozisi

INTRODUCTION

Although dysphagia and cervical spondylosis are both common presenting problems, theyare often unrelated. A review of the literature showed that dysphagia caused by this type of bony change is relatively rare, with fewer than 200 cases reported to date (4,11). The association of these two problems was first reported in 1926 by Mosher (4). In 1950, Forestier described a clinical manifestation that was characterized by spinal rigidity, and in the 1970s this was named "diHuse idiopathic skeletal hyperostosis" (DISH). Most reported cases of spinal

(DISH), disfaji, Forestier hastaligi, spinal osteofitosiz

osteophyte-related patients (4,10).

dysphagia

have been in DISH

The diagnostic criterias for DISH, also known as Forestier's disease, are as follows: 1) flowing calcification/ossification along the anterola teral aspect of four contiguous vertebral bodies 2) relative preservation of intervertebral disc height in affected areas and 3) absence of apophyseal joint ankylosis and sacroihac joint selerosisl fusion (lO). The etiology is unknown, but the re is no relationship between this condition and ankylosing spondylitis, degenerative disc/joint disease, or rheumatoid arthritis. The 117

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1001

majority of cases are asymptomatic. The overall prevalence of DISH is higher than would be expected in the predominantly white population over 50 years of age, and the frequency is lower in the AfricanAmerican, Native-American, and Asian populations, suggesting that there may be a genetic origin (13). A report daimed that Middle Eastem populations exhibit less dysphagia symptomatology than United States and European populations, which have higher frequencies of DISH (1). it is uncommon to diagnose this disease in patients less than 50 years of age. The prevalence ilKreases with age, to a maximum of 28% in individuals 70 to 80 years of age. AIso, there is a gender bias towards men in those affected, with a male:female ratio of 2:1 (4). CASE

DESCRIPTION

A 68-year-old man presented with the complaint of painful swallowing of 2 years' duration. The problem had gradually worsened over the previous 6 months, and he had recently developed problem s swallowing even liquid food. The patient

Figure 1: A lateral plain film of the cervical spine shows a large anterior cervical osteophyte extending from C2 to CS. The trachea is displaced ventrally.

118

Be/eli: Verlei"'ngciiic Dys/'Iliigin

had lost approximately 10 kg ofbody weight during the past 6 months. His laboratory findings were unremarkable, apart from a slightly elevated bloodglucose level of 150 mg/ dL. Gastrointestinal specialists attempted esophagoscopy assessment, but this was unsuccessful due to a high-Ievel obstruction of the esophagus. A plain x-ray of the cervical region shm"1ed extensive cakification along the anterior aspect of the cervical column from the C2 to CS vertebrae (Figure 1). A barium-swallow esophagogram revealed mechanical obstruction caused by this same lesion, but showed no other pathology (Figure 2). Cervical magnetic resonance imaging (MRI) revealed details of the lesion and the displacement of the surrounding soft tissue (Figure 3). There were no significant degenerative osteophytic changes within the spinal canal or the foraminae of the cervical vertebrae, nor was any calcification of the posterior longitudinal ligament. rlain radiographic screening of other parts of the skeletal system revealed no additional osteophytic degeneration.

Figure 2: A lateral-view demonstrates obstruction.

bariuin-swa!low a high-Ievel

esophagogram esophageal .

Turkisli

NeiiroS1lrgery

11: 117 -

120, 2001

Bekii: Vertebrngeiiic

Dysplrngin

individuals with dysphagia who have cervical osteophytes larger than 10 mm diameter (ll). Regarding other regions, thoracic spondylosis has been cited as a cause of myelopathy, Homer's syndrome, radiculopathy, thoracic outlet syndrome, obstructive pneumonia, and esophageal food impaction. Similar to the above situation, symptoms are more likely to arise in patients with DISH, who have large bridging osteophytes. Lumbar osteophytes in DISH patients have be en implicated in lumbar spinal stenosis and obstruction of the inferior vena cava. DISH is also associate d with diabetes mellitus, with extraosseous cakification in the tendons and peripheralligaments, and with heel, shoulder, and elbow pain. Bony fractures are also a feature in this patient group (6).

Figure 3: Sagittalcervical MRI reveals the same pathology that was detected on plain radiographs, but without pathologic enhancement. There are no obvious osteophytic changes in the spinal canal or vertebral foraminae.

Since the patient was having severe problem of swallowing even liquid food, and in light of the progression of the disease, we advised that the compressing mass should be surgically removed. However, he refused surgical treatment and was placed on a specialliquid diet. Six months later, his weight had stabilized and he was able to swallow the prescribed recipe. DISCUSSION DISH is a common skeletal condition that can affect the entire spinal column, but the two most frequently affected regions are the thoracic and cervical spine, respectively (LO). Cervical spondylosis is recognized as a rare cause of dysphagia, aspiration pneumonia, myelopathy, cervical root compression, upper airway obstruction, dysphonia, and vertebral artery compression. Symptoms are particularly prevalent in patients with DISH, who may have large cervical osteophytes. Aspiration is common in

The diagnosis of DISH is based on radiological assessment of the thoracic spine, although extraspinal involvement can also help identify the disease (7). Plain x-rays are the first step in patient evaluation. The spinal radiological manifestations of DISH are characterized by extensive ossification of the anterolateral aspect of the vertebral bodies, with relative preservation of disc height. Bridging ossification is most common in the thoracic spine, and non-bridging ossification is characteristic of cervical and lumbar involvement. Ossification of the posterior longitudinal, intraspinous, nuchal, and flavum ligaments has been reported as well (7). The next diagnostic step may be MRI or CT, which allow the examiner to investigate the pathology in detail and plan the surgical approach. A barium-swallow esophagogram is indicated in cases of cervical involvement to assess esophageal function. When a patient known to have a cervical vertebral bone spur complains of dysphagia, it must be determined whether the protuberance is the singular cause of the dysphagia, a contributing factor, or whether it plays any role at all. Alternative causes of dysphagia must be considered, including neurological diseases (stroke, Parkinson' s disease, and amyotrophic lateral selerosis) andi or mechanical obstruction due to head/neck cancer, mediastinal masses, Zenker' s diverticulum, esophageal webs, and stricture or cancer of the esophagus (8). In addition, gastroesophageal reflux can cause symptoms similar to those seen with cervical bony protuberances. Asymptomatic patients with cervical vertebral bony changes or cases where a problem is detected incidentally may be approached conservatively. The 119

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natural course of the disease is slow, andsurgery is associated with significant morbidity and mortality. Sometimes it is difficult to intubate these individuals due to their rigid cervical spine, and fiberscope guidance can help in these cases. Some elinicians prefer to intubate while the patient is awake (9). The most widely accepted treatment for cases of severe dysphagia is surgical removal of the compressing mass, but the problem does not always resolve immediately af ter the operation. Some authors have claimed that the dysphagia is caused by a combination of mechanical pressure, chronic esophageal wall inflammation, and dysfunction of the nerve supply to the wall due to stretching. McCafferty et aL. (8) reported that their patients' dysphagia had continued lang af ter surgical treatment, whereas others have found that the problem resolved immediately (3). Another issue of debate regarding surgical management of these cases is the need for spinal stabilization af ter the mass removed. Some authors have pointed out that the process is slow, the spine is rigid (lO), and elderly people are at greater risk of developing problems during or af ter any long surgery. These investigators believe that cervical fusion does not help these individuals, and that the restricted neck motion af ter fusion may cause them to suffer even more (8). In contrast, most published data have shown that cervical fusion af ter osteophyte removal is essential for stabilization (2,3,4). Our opinion is that the decision to perform any fusion procedure should be made on a case-by-case basis. Today, various disciplines are attempting to manage this pathology with significantly different approaches. Some orthopedic surgeons and ear-nosethroat specialists do not hesitate to treat cervical osteophytic lesions without neurosurgical consultabon (5,12), but their surgical methods are not necessarily what a neurosurgeon would advise. Neurosurgeons frequently dea with problems in the cervical spine, and are very experienced with the pathologies that affect all structures in this region. In our opinion, patients with cervical vertebral osteophytes who are surgical candidates should always be consulted by a neurosurgeon.

i

Correspondence:

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Beleii: Verte/Jrngeiiic Dysphasia

2001

Deniz Belen, SSK Ankara Egitim Hastanesi, II. Nörosirürji Klinigi, Diskapi 06150, Ankara, Turkey e-mail: [email protected]

REFERENCES 1. EI-Gad A, Khater R: Diffuse idiopathie skeletal hyperostosis (DISH). A clinical study of the disease pattem in Middle Eastem populations. J Rheumatol 11 (6): 804-807, 1984.

2. Epstein NE: Simultaneous eervieal diffiise idiopathie sk eleta1hyperostosis and ossifieation of the posterior longitudinal ligament resulting in dysphagia or myelopathy in two geriatrie North Amerieans. Surg Neurol 53: 427-431, 2000 3. Goffin J, Calenbergh FV: Forestier's Disease. J Neurosurg 85: 524-525, 1996. 4. Granvil1e LL Musson N, Altman R, Silverman M: Anterior eervieal osteophytes as a eause of pharyngeal stage dysphagia. J Am Geriatr Soc 46(8): 1003-1007, 1998

5. Grasshoff H, Motsch C, Mahfeld K: Vertebragenic dysphagia. Zentralbl Chir 124: 1041-1044, 1999 6. Le Hir PX, Sautet A, Le Gars L, Zeitoun F, Tubiana JM, Arrive L, Laredo JD: Hyperextension vertebral body fraetures in diffuse idiopathic skeletal hyperostosis: a caiise of intravertebral fluidlike eolleetions on MR imaging. AJR 173: 1679-1683, 1999 7. Mata S, Chhem K, Foertin PR, Joseph L, Esdaile JM: Comprehensive radiographic evaiuation of diffuse idiopathic skeletal hyperostosis: development and interrater reHability of a scoring system. Semin Arthritis Rheum 28: 88-96, 1998 8. McCafferty RR, Harrison MJ, Tamas LB, Larkins MV: Ossification of the anterior longitudinal ligament and Forestier's disease: an analysis of seven cases. J Neurosurg 83: 13-17, 1995 9. Palmer JH, Ball DR: Awake intubation with the intubating laryngeal mask in a patient with diffuse idiopathic skeletal hyperostosis. Anaesthesia 55(1): 7074,2000.

10. Rotes-Querol J: Clinical manifestations of diffiise idiopathic skeletal hyperostosis (DISH). British J Rheumatol 35: 1193-1196, 1996 11. Strasser G, Schima W, Schober E, Pokieser P, Kaider A, Denk DM: Cervieal osteophytes impinging on the pharynx: importanee of size and concurrent disorders for development of aspiration. AJR 174(2): 449-453, 2000

12. Up pal S, Wheatley AH: Transpharyngeal approach for the treatment of dysphagia diie to Forestier's disease. J Laryngol Otol 113(4): 366-368, 1999. 13. Weinfeld RM, Olson PN, Maki DD, Griffiths HJ: The prevalence of diffuse idiopathic skeletal hyperostosis (DISH) in two large American Midwest metropolitan hospital populations. Skeletal Radiol 26: 222-225, 1997

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